Spinal thoracic extradural angiolipoma manifesting as acute onset of paraparesis: Case report and review of literature
- Neurosurgeon, Sao Paulo Federal University, Sao Paulo, Brazil
- Neurologist, Hospital das Clinicas Sao Paulo, Sao Paulo, Brazil
- Neurosurgeon, Villa Lobos Hospital, Sao Paulo, Brazil
- Neuropathologist, Sao Paulo Federal University, Sao Paulo, Brazil
Franz Jooji Onishi
Neuropathologist, Sao Paulo Federal University, Sao Paulo, Brazil
DOI:10.4103/sni.sni_467_16Copyright: © 2017 Surgical Neurology International This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 3.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.
How to cite this article: Franz Jooji Onishi, Flavio Augusto Sekeff Salem, Diogo Luis de Melo Lins, Rafi Felicio Bauab Dauar, João Norberto Stavale. Spinal thoracic extradural angiolipoma manifesting as acute onset of paraparesis: Case report and review of literature. 18-Jul-2017;8:150
How to cite this URL: Franz Jooji Onishi, Flavio Augusto Sekeff Salem, Diogo Luis de Melo Lins, Rafi Felicio Bauab Dauar, João Norberto Stavale. Spinal thoracic extradural angiolipoma manifesting as acute onset of paraparesis: Case report and review of literature. 18-Jul-2017;8:150. Available from: http://surgicalneurologyint.com/surgicalint-articles/spinal-thoracic-extradural-angiolipoma-manifesting-as-acute-onset-of-paraparesis-case-report-and-review-of-literature/
Background:Angiolipomas are benign tumors most commonly found in the thoracic spine. They are composed of mature adipocytes and abnormal vascular elements that usually present with a slowly progressive course of neurological deterioration.
Case Description:A 35-year-old female, with a prior history of back pain, acutely developed paraparesis. When the thoracic magnetic resonance imaging (MRI) revealed a dorsal epidural mass at the T3-T5 level, she underwent a laminectomy for gross total excision of the lesion that proved to be an angiolipoma. On the second postoperative day, the patient was again able to ambulate.
Conclusion:The angiolipomas of spine are rare causes of spinal cord compression, and those presenting with acute neurological deficits should be immediately treated.
Keywords: Angiolipoma, extradural spinal tumor, spinal cord compression, spinal tumor
Angiolipomas are benign tumors consisting of mature fat cells and proliferating abnormal blood vessels. They generally occur in the subcutaneous tissue of the trunks and limbs. They can be further categorized into two subtypes – noninfiltrating (more common) and infiltrating. They account for 0.004–1.2% of all spinal axis tumors and 2–3% of extradural spinal lesions.[
A 35-year-old female, with 4 weeks of bilateral lower limb numbness, acutely presented with 24-hour evolution of an acute paraparesis, accompanied urinary retention. Physical examination revealed 4/5 motor strength in both the legs, diffuse lower extremity hyperreflexia, and superficial hypoesthesia below the T4 level.
The magnetic resonance imaging (MRI) scan showed a large fusiform dorsal lesion measuring 8.0 cm × 2.8 cm × 1.1 cm compressing the thoracic spinal cord extending over the T3-T5 levels [
Sagittal MRI images showing heterogeneous epidural posterior mass compressing thoracic cord over three body segments between T3-T5. (a) T1-weighted MRI shows inhomogeneous iso to hypointensity mass; (b) Post-contrast T1-weightted MRI shows enhancing mass; (c) T2-weighted MRI shows a hyperintense tumor
A laminotomy of T3-T5 was performed under general anesthesia with intraoperative neurophysiological monitoring. The posterior epidural space was filled with a fatty, highly vascular brown-pink mass that was extremely hemorrhagic. It was easily mobilized away from the compressed dura, and a gross total resection was accomplished (e.g., in one piece). There were no changes in the intraoperative motor evoked potentials [
On postoperative day 2, the patient was able to ambulate, and she had fully recovered within 2 weeks. After 2 years, the patient remains asymptomatic and shows no signs of spinal deformity. There is also no evidence of radiographic recurrence.
The analysis of the tumor revealed a lesion with adipocytes and capillary sized vessels [
These lesions consist of varying portions of mature fat cells and abnormal capillary, sinusoidal, venous, or arterial vascular elements. The ratio of fat to vessels is variable and ranges from 1:3 to 2:3. Tumors with an abundance of smooth muscle proliferation are further subclassified as angiomyolipomas. Angiolipoma generally arise from the dorsal aspect of spinal canal at thoracic levels compressing the spinal cord and causing symptoms.[
The origin and pathogenesis of angiolipomas are through to arise from pluripotential mesenchymal stem cells with secretory activity. Early inclusion of pluripotent stem cells during the developmental ossification of neural arch is believed to be a prerequisite for spinal angiolipoma formation. Degenerative changes may be presenting in some longstanding cases but malignant transformation and neural tissue infiltration have never been reported.[
Angiolipomas predominantly occur in females (female:male ratio = 22:17) in their fifties who typically suddenly develop paraparesis. The rapid onset of symptoms in usually due to vascular factors; e.g., anomalous vessels, intralesional thrombosis, and hemorrhage or steal phenomena.[
Demyelinating disease must be considered among the differential diagnoses as occasionally these lesions present with relapsing/remitting course.[
MRI, T1-weighted images typically demonstrate a high signal accompanied by an inhomogeneous mass. On T2-weighted images, the signal intensity seems to be similar to adipose tissue. The lesion markedly enhances with gadolinium administration [
Treatment and prognosis
Spinal angiolipomas are treated exclusively by surgical removal, and most may be completely excised with good clinical results (e.g., normal postoperative examinations).[
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Conflicts of interest
There are no conflicts of interest.
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